Hemophagocytic Lymphohistiocytosis in Children with Visceral Leishmaniasis

Blázquez‐Gamero, Daniel; Domínguez‐Pinilla, Nerea; Chicharro, Carmen; Negreira, Sagrario; Galán, Pilar; Pérez-Gorricho, Beatriz; Calvo, Cristina; Prieto, Luís; Parte, María De la; Otheo, Enrique; Vivanco, José Luis Melgarejo; Ruiz–Contreras, Jesús

doi:10.1097/inf.0000000000000685

Cited by 20 publications

(27 citation statements)

References 12 publications

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“…In our series, the patients had fever, splenomegaly with/without hepatomegaly, pancytopenia, elevated liver enzymes, with similar frequency reported previously (15,18,19). In previous reports, VL is found to be related to HLH (14,27). In our series, HLH was diagnosed in only one patient; however, the other patients were not investigated for HLH.…”

Section: Discussionsupporting

confidence: 88%

Multiple Nodular Lesions in Spleen and Liver in Visceral Leishmaniasis

Kutlu

Herek

et al. 2019

Infect Dis Clin Microbiol

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Objective:We aimed to describe the clinical, laboratory, and radiological characteristics of visceral leishmaniasis (VL) in our region to find additional clues that can help diagnosis. Materials and Methods:All the patients with VL who admitted to our clinic between January 2010 and January 2018 were reviewed retrospectively. The patients with symptoms and signs suggestive of VL and a positive indirect fluorescent antibody test (IFA) or rapid diagnostic test, and/or direct parasitology consisting of the presence of amastigotes of Leishmania in bone marrow aspirate were included in the study. We collected the demographic and clinical features, laboratory, and radiological information from the hospital database. Results:We present ten (seven males and three females) cases of VL, with the mean age of 53 (range of 31-75). Serological tests such as IFA or rapid diagnostic tests were positive in all patients. Amastigotes of Leishmania were present in only three of eight patients who underwent bone marrow biopsy. Abdominal magnetic resonance imaging or computed tomography revealed that all patients (10/10) had splenomegaly, eight of 10 patients had hepatomegaly, and seven patients had intraabdominal lymphadenopathy. Seven patients had multiple hypodense nodular lesions in the spleen with homogeneous parenchyma. Two patients had nodular lesions in the liver. Two patients died because of multiorgan failure before completing their treatment. The remaining patients completed liposomal amphotericin B treatment and cured.Conclusion: VL should be suspected in the diagnosis of the patients, who had long-term fever, splenomegaly, pancytopenia associated with multiple nodular lesions in the spleen with/without liver involvement.

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Section: Discussionsupporting

confidence: 88%

Multiple Nodular Lesions in Spleen and Liver in Visceral Leishmaniasis

Kutlu

Herek

et al. 2019

Infect Dis Clin Microbiol

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“…A diagnosis is made when >5/8 of the HLH criteria are met4 5 as in our case (see box 1). Hyperferritinaemia>10 000 is the most sensitive marker 1…”

Section: Discussionmentioning

confidence: 99%

“…The pathogenesis of sHLH remains unclear. Both the dysregulation of CD8 +T lymphocytes and non-malignant proliferation of macrophages lead to uncontrolled hypercytokinaemia 1 7 8. This, in turn, causes migration of these inflammatory cells and organ infiltration causing cell necrosis and multi-organ failure.…”

Section: Discussionmentioning

confidence: 99%

“…One such diagnosis is haemophagocytic lymphohistiocytosis (HLH) which is a rare but life-threatening condition characterised by inflammation and immune hyperactivation causing uncontrolled cytokine release. The clinical picture initially resembles sepsis, but then progresses in an insidious fashion with multi-system involvement including: pancytopenia, hepatosplenomegaly and hyperferritinaemia 1 2. There are two types of HLH: familial HLH (fHLH) and secondary HLH (sHLH).…”

Section: Introductionmentioning

confidence: 99%

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Haemophagocytic lymphohistiocytosis complicating visceral leishmaniasis in the UK: a case for detailed travel history, a high index of suspicion and timely diagnostics

Johnson¹,

Gilmour

Samarasinghe

et al. 2019

BMJ Case Rep

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A 4-month-old male infant presented acutely unwell with fever. He was initially treated for sepsis but failed to improve with IV broad spectrum antibiotics. Haemophagocytic lymphohistiocytosis (HLH) was diagnosed due to his fever, pancytopenia, splenomegaly, hypertriglyceridaemia, hypofibrinogenaemia and significant hyperferritinaemia. An array of differentials for HLH including both immunological and infectious causes were considered and excluded. He had travelled to Madrid, and hence visceral leishmaniasis (VL) was suspected, but was not confirmed on the initial bone marrow aspirate (BMA) microscopy or culture. He improved with empirical treatment with dexamethasone and liposomal amphotericin B. VL was later confirmed on BMA PCR. He made a good recovery and remained well at 12 month follow-up. Non-endemic countries need rapid and sensitive VL diagnostics. A thorough travel history and high clinical index of suspicion are necessary to avoid the pitfall of treatment with intense immunosuppression recommended in treatment guidelines for HLH.

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“…Leishmania parasites have been found to be the most common protozoan trigger of acquired HLH. In a multicenter prospective study conducted to determine the frequency of HLH syndrome in children with VL, ten children out of 24 (41 %) with VL developed HLH syndrome [4]. HLH incidence in European adult population is about 1/800,000/year, with a reported prevalence of parasitic infections of 2.4 % (53 out of 2197 subjects), and of Leishmania spp.…”

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confidence: 99%

Visceral leishmaniasis, hypertriglyceridemia and secondary hemophagocytic lymphohistiocytosis

et al. 2016

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Visceral leishmaniasis (VL), a protozoan disease, is 100 % fatal if left untreated. Anemia is common in VL which plays a role in expression of clinically overt VL disease. Laboratory clues are scarce for strengthening clinical suspicion for severity in VL. Hypertriglyceridemia has emerged as a new concept for the diagnosis and prognosis in VL. The present study is aimed at correlating the magnitude of hypertriglyceridemia with the severity in VL. Materials and methods: A retrospective case–control study was conducted between January 2012 to December 2013 among 124 patients coming for treatment from VL endemic areas, who had fever of more than 15 days and did not respond to antimalarials and antibiotics. The parasitologically confirmed VL cases (n = 87) were categorized as mild/moderate (n = 60) and severe (n = 27) groups according to WHO classification for anemia and parasite burden. Serum triglycerides were assayed in VL groups along with controls (n = 37). Results: Serum triglyceride level was significantly higher in VL than controls [mean values were 173.50 ± 47.67 versus 127.1 ± 53.79 mg/dl, respectively (p < 0.0001)]. Triglyceride level was significantly higher in severe than in mild/moderate group of VL [211.3 ± 50.2 mg/dl versus 134 ± 45.09 mg/dl, respectively (p < 0.0001)]. Hypertriglyceridemia (>161.7 mg/dl) was noted in all severe VL patients, compared to 31.66 % of mild or moderate group (p < 0.0001). There was no significant difference between mild/moderate VL and controls. Conclusions: It is hypothesized that hypertriglyceridemia could be of additional diagnostic benefit to assess the probability and severity of VL in endemic areas. © 2015, Springer-Verlag Berlin Heidelberg

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Hemophagocytic Lymphohistiocytosis in Children with Visceral Leishmaniasis

Cited by 20 publications

References 12 publications

Multiple Nodular Lesions in Spleen and Liver in Visceral Leishmaniasis

Multiple Nodular Lesions in Spleen and Liver in Visceral Leishmaniasis

Haemophagocytic lymphohistiocytosis complicating visceral leishmaniasis in the UK: a case for detailed travel history, a high index of suspicion and timely diagnostics

Visceral leishmaniasis, hypertriglyceridemia and secondary hemophagocytic lymphohistiocytosis

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